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| CASE REPORT |
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| Year : 2022 | Volume
: 60
| Issue : 4 | Page : 318-320 |
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Asymmetric and incomplete vogt Koyanagi – Harada syndrome: Patient's perceptive dilemma
Priyanka H Gandhi, Syed Faraaz Hussain, Abha Shah, Yogya Reddy
Department of Ophthalmology, Mahatma Gandhi Missions Institute of Health Sciences, Navi Mumbai, Maharashtra, India
| Date of Submission | 13-May-2022 |
| Date of Decision | 29-Aug-2022 |
| Date of Acceptance | 02-Sep-2022 |
| Date of Web Publication | 19-Dec-2022 |
Correspondence Address:
Priyanka H Gandhi
Department of Ophthalmology, MGM Hospital, Kamothe, 711, PG Hostel, MGM Campus, Kamothe.410 209, Navi Mumbai, Maharashtra
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/tjosr.tjosr_49_22
Vogt–Koyanagi–Harada (VKH) is a rare autoimmune multisystem disease. Here, we report a case of VKH with asymmetric involvement and the importance of patient counselling and a comprehensive approach. A 45-year-old female patient went to tertiary care with left-sided headache and sudden diminution of vision in the left eye (LE) since 1 day. She was advised lumbar puncture but became apprehensive and came to us with visual acuity (VA) 6/6 in the right eye (RE) and HMCF PL+, PR accurate in LE. LE showed lid oedema, mild conjunctival congestion, vitritis, disc oedema, choroidal detachment and exudative retinal detachment (ERD) involving the macula. The patient was suspected to have incomplete VKH. Neurologic, Ear, Nose, Throat (ENT) and dermatology consults were taken. The patient was counselled about the systemic issue, the possibility of other eye involvement later and the need for a team-based approach. The patient was started on steroids. At 6 months, VA in RE - 6/36 and in LE - 6/60, RE showed a pocket of ERD. Asymmetric involvement of both eyes is an infrequent presentation highlighting the necessity of a comprehensive approach along with diligent patient counselling for allaying fears.
Keywords: Multidisciplinary, patient counselling, VKH
How to cite this article:
Gandhi PH, Hussain SF, Shah A, Reddy Y. Asymmetric and incomplete vogt Koyanagi – Harada syndrome: Patient's perceptive dilemma. TNOA J Ophthalmic Sci Res 2022;60:318-20 |
How to cite this URL:
Gandhi PH, Hussain SF, Shah A, Reddy Y. Asymmetric and incomplete vogt Koyanagi – Harada syndrome: Patient's perceptive dilemma. TNOA J Ophthalmic Sci Res [serial online] 2022 [cited 2023 Feb 8];60:318-20. Available from: https://www.tnoajosr.com/text.asp?2022/60/4/321/364249 |
| Introduction | |  |
Vogt–Koyanagi–Harada syndrome (VKH) is a autoimmune multisystem disease characterized by bilateral granulomatous uveitis associated with auditory, neurological and dermatological manifestations. Diagnostic criteria for VKH syndrome by The American Uveitis Society included no history of ocular trauma or procedure and at least one finding in three, out of the following: 1) posterior uveitis, including exudative retinal detachment, disk oedema and sunset glow fundus; 2) bilateral chronic iridocyclitis; 3) neurologic signs: tinnitus, meningismus; 4) cutaneous findings of alopecia, poliosis and/or vitiligo.[1] It is more common in the adult population and has a predilection for the female gender.[2] The distinctiveness of our case was the asymmetric involvement of both eyes highlighting the necessity of a comprehensive approach along with diligent patient counselling for allaying fears.
| Case History | | |
A 45-year-old female patient presented to ophthalmology service with the chief complaint of left-sided temporal headache since 2 days, giddiness since 3 days followed by a sudden and painful diminution of vision in the left eye since 1 day. She underwent left ear surgery 6 years ago. There was no history of spectacle usage, ocular trauma, ocular procedure or any other systemic illness.
On examination, visual acuity for distant vision was noted to be 6/6 in the right eye and HMCF PL positive, PR accurate in the left eye. Near vision was noted to be N6 in the right eye, whereas less than N36 in the left eye. Colour vision in the right eye was 17/17 plates and Amsler's test was normal. No reading could be obtained from the left eye for colour vision and Amsler's test. Extraocular movements were free, full and painless in all gazes. On anterior segment examination of the left eye, lid oedema, mild conjunctival congestion, clear cornea, normal depth anterior chamber, clear lens and 4 mm pupil sluggishly reacting to light were seen. The anterior segment of the right eye was within normal limits. Posterior segment examination of the left eye revealed vitritis, pale retina, disc oedema, choroidal detachment and exudative retinal detachment involving the macula [Figure 1] and [Figure 2]. Posterior segment examination of the right eye was within normal limits. Intraocular pressure (IOP) was recorded as 18 mmHg in the right eye and 20 mmHg in the left eye using the Goldmann applanation tonometer. Optical coherence tomography (OCT) was done, which revealed pigment epithelial detachment (PED) in the right eye and exudative detachment in the left eye [Figure 3] and [Figure 4]. MRI brain + orbit was done, which was suggestive of left optic neuritis, posterior scleritis and periscleral cellulitis. | Figure 2: Fundus photography of the left eye at presentation showing vitritis, pale retina, disc oedema, choroidal detachment and exudative retinal detachment involving the macula
Click here to view |
The patient was referred to the Medicine, ENT and Dermatology department to rule out other systemic manifestations of VKH. A lumbar puncture was done, which revealed lymphocytic meningitis. Pure tone audiometry revealed right-sided high-frequency moderate sensorineural hearing loss (SNHL) and left-sided moderate SNHL. No dermatological lesions were found.
The patient was started on intravenous methylprednisolone for 5 days followed by oral steroids with gradual tapering. E/d Bidin-T BD, E/d moxifloxacin four times, E/d CMC four times and E/d homatropine BD were advised to be applied topically in the left eye.
She was advised to follow up regularly and at each visit, the findings were noted.
At 4 months, visual acuity for distant vision in the right eye was 6/36 improving to 6/12p with pinhole and in the left eye was 6/60 improving to 6/18p. Anterior segments of the right and left eyes were within normal limits. Fundus examination of the right eye was within normal limits and the left eye revealed pale disc, the presence of chorioretinal atrophic patches implying reduced inflammation [Figure 5]. IOP was recorded as 14 mmHg in both eyes using the Goldmann applanation tonometer. She was on oral prednisolone 40 mg OD. All topical drugs were stopped. | Figure 5: Fundus photography of the left eye at follow-up after 4 months showing pale disc and chorioretinal atrophic patches
Click here to view |
| Discussion | | |
Patients with VKH present with varied ocular manifestations such as iridocyclitis, diffuse choroidal thickening and hyperaemia of the optic disc. Headache is the main neurological manifestation and auditory changes may present with tinnitus and hearing loss along with skin manifestations such as alopecia, poliosis and vitiligo.[3] The mainstay of treatment of VKH is corticosteroid therapy in the acute phase along with an additional immunosuppressive agent.[4]
Shrestha et al.[2] reported four cases of VKH with typical features of VKH with bilateral involvement in all the cases.
Agrawal et al.[5] reported two cases that indicated the clinical and angiographic features characteristic of VKH with unilateral involvement.
Dhami et al.[6] reported a case in which atypical unilateral VKH disease mimicked central serous retinopathy such as a retinal picture.
Usui et al.[7] reported three cases with clinical and laboratory features of VKH disease with unilateral eye involvement.
In our case, the patient presented with unilateral ocular complaints and was unaware of the multisystem involvement nature of the disease. Through proper counselling, the patient was encouraged to be compliant with the comprehensive management for optimal care.
Timely identification and comprehensive management along with diligent patient counselling can minimize ocular morbidity and reduce the risk of involvement of the other eye in unilateral cases of VKH.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the legal guardian has given her consent for images and other clinical information to be reported in the journal. The guardian understands that her names and initials will not be published and due efforts will be made to conceal the patient's identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | |
| 2. | Shrestha P, Sharma S, Kharel R. Vogt-Koyanagi-Harada Disease: A Case Series in a Tertiary Eye Center. Case Rep Ophthalmol Med 2021;2021:8848659. doi: 10.1155/2021/8848659. PMID: 33552608; PMCID: PMC7847319.  |
| 3. | Moorthy RS, Inomata H, Rao NA.Vogt-Koyanagi-Harada syndrome.Surv Ophthalmol 1995;39:265-92. |
| 4. | Baltmr A, Lightman S, Tomkins-Netzer O. Vogt-Koyanagi-Harada syndrome-current perspectives. Clin Ophthalmol 2016;10:2345-61. |
| 5. | Agrawal A, Biswas J. Unilateral Vogt-Koyanagi-Harada disease: Report of two cases. Middle East Afr J Ophthalmol 2011;18:82-4. [ PUBMED] [Full text] |
| 6. | Dhami, Abhinav & Dhami, Gobinder. Unilateral and Atypical Presentation of Vogt-Koyanagi-Harada Syndrome: A Case Report. J Clin Diagn Res 2019;13. 10.7860/JCDR/2019/40575.12797. |
| 7. | Usui Y, Goto H, Sakai J-i, Takeuchi M, Usui M, Rao NA. Presumed Vogt Koyanagi Harada disease with unilateral ocular involvement: Report of three cases. Graefes Arch Clin Exp Opthalmol 2009;247:1127-32. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
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